European Child & Adolescent Psychiatry (2019) 28:57–68

https://doi.org/10.1007/s00787-018-1187-z

ORIGINAL CONTRIBUTION

Combined habit reversal training and exposure response prevention

in a group setting compared to individual training: a randomized
controlled clinical trial
Judith B. Nissen1,3   · Martin Kaergaard1 · Lisbeth Laursen1 · Erik Parner2 · Per Hove Thomsen1,3

Received: 26 February 2018 / Accepted: 23 June 2018 / Published online: 28 June 2018
© The Author(s) 2018

Abstract
Chronic tic disorders may have a huge influence on quality of life. Habit reversal training (HRT) and exposure response
prevention (ERP) are effective treatments. In a blinded assessed, open trial, this study evaluates the effectiveness of a newly
developed Scandinavian tic treating manual designed to treat adolescents with a chronic tic disorder, combining HRT and
ERP. The study compared the efficacy of treatment based on the same manual delivered either individually or in groups. The
study was an open randomized controlled clinical trial in which adolescents were randomized to either individual or group
therapy. Both therapies included nine sessions. The parents were offered group-based psycho-education. The exclusion criteria
were chosen to design a study that would be close to clinical practice. This is the first Scandinavian study that examines the
effectiveness of a treatment manual combining HRT and ERP delivered in an individual and group setting. The study showed
a significant reduction of the Total Tic score on the Yale Global Tic Severity Scale both in the individual (effect size 1.21)
and group setting (effect size 1.38). A total of 66.7% of participants were considered responders. There was no statistical
significant difference between the individual and group setting apart from the functional impairment score. The reductions
were comparable with those shown in other studies. The participants applied both HRT and ERP, and the majority (36/59)
reported an increased post-treatment experience of control. The newly designed Scandinavian manual was equally effective
in the individual and group setting with effect sizes comparable with those shown in other studies.

Keywords  Tourette syndrome · Pediatric · Habit reversal training · Exposure response prevention · Group · Manual

Abbreviations
TS Tourette syndrome
HRT Habit reversal training
ERP Exposure response prevention
* Judith B. Nissen CBIT Comprehensive behavioral intervention for
[email protected]
tics
Martin Kaergaard ADHD Attention deficit hyperactivity disorder
[email protected]
K-SADS-PL Schedule for Affective Disorders and
Lisbeth Laursen Schizophrenia for School-Age Children—
[email protected]
Present and Lifetime version
Erik Parner CBCL Child Behavior Checklist
[email protected]
SDQ Strengths and Difficulties
Per Hove Thomsen SP Sensory profile
[email protected]
YGTSS Yale Global Tics Severity Scale
1
Center for Child and Adolescent Psychiatry, Aarhus SCARED Screen for Child Anxiety Related
University Hospital Risskov, Risskov, Denmark Emotional
2
Section of Biostatistics, Department of Public Health, Aarhus MFQ The Mood and Feelings Questionnaire
University, Aarhus, Denmark PUTS Premonitory Urge Scale
3
Institute of Clinical Medicine, Health, Aarhus University, BATS Beliefs About Tics Scale
Aarhus, Denmark

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58 European Child & Adolescent Psychiatry (2019) 28:57–68

Background for prolonged periods of time, and they are instructed to

practice suppressing or resisting the tic symptoms. Unlike
Chronic tic disorders (CTD), including Tourette syndrome, HRT, no competing response is trained. In the manual,
are neuropsychiatric, neurodevelopmental disorders Verdellen et  al. [18] described 12 structured sessions
with a prevalence of 0.5–1%. Tourette syndrome affects using ERP. Furthermore, the manual described ten ses-
mostly males (4:1) and has an age of onset between 6 and sions of HRT. In a single randomized controlled study, it
8 years, where motor tics often precede the vocal tics [1, was shown that both treatment modalities resulted in a sig-
2]. Though fluctuating in tic intensity, severity and locali- nificant effect measured by YGTSS, tic frequency counted
zation, tic disorders may have a huge impact on quality of in the institute and at home. Effect sizes of 1.42 and 1.06
life and daily living [3, 4]. This places high demands on were comparable for ERP and HR, respectively [19].
the availability of effective treatments. Both manuals for HRT/CBIT and ERP [11, 18] describe
European guidelines recommend a behavioral and psy- structured sessions focusing on either HRT or ERP for indi-
chosocial intervention as the primary treatment of CTD in vidual therapy.
children and adolescents [5]. Pharmacological treatment Group therapy has been shown to be effective in rela-
indicated by the tic disorder is viewed as a supplemental tion to other psychiatric diagnoses, which is not least known
treatment [5–7]. Therapeutic treatments of CTD include from cool kids sessions in relation to anxiety disorders in
habit reversal treatment (HRT) and exposure and response children and adolescents [20]. Children and adolescents
prevention (ERP) [5]. often ask for contact to peers with comparable problems.
Habit reversal (HR) was introduced in 1973 by Azrin Only one study has examined the effect of HRT in a group
et al. [8] and later on as part of Comprehensive Behavioral setting compared with group educational therapy [21]. Both
Intervention for Tics (CBIT) [9–11]. The premonitory urge group interventions showed a positive effect. However, the
is a sensory phenomenon preceding the tic symptom and reduction of motor tics was larger in the HRT group. Still, a
is often described as an amplifier of tics even though no direct comparison of the effect of individual treatment com-
relation has been shown between reductions in premoni- pared to group therapy is lacking.
tory urge scores and treatment outcome [12] or the ability Furthermore, children and adolescents with CTD differ
to suppress tics [13]. In HRT, a competing response is not least in relation to the occurrence of comorbid condi-
trained which helps the child to endure the internal pres- tions, where almost 90% experience another psychiatric or
sure made by the premonitory urge [10, 11]. Woods et al. somatic condition [22, 23]. Thus, it is still unknown whether
[11] presented a structured treatment manual describing age, tic severity, and comorbid conditions may alter or influ-
eight sessions delivered individually over a period of ence a child’s ability to engage in therapy or may influence
10 weeks, supplemented with three booster sessions. Two the preference of one therapy setting over another. Clinical
randomized controlled trials have examined the effect of effectiveness of a combined treatment offering training in
HRT/CBIT compared to a control group in children/ado- both HRT and ERP is still unknown.
lescent populations. Both studies showed that treatment The aim of this blindly assessed open trial was to evaluate
with HRT/CBIT significantly reduced the tic intensity as a the effectiveness of a newly designed Scandinavian manual
total score with effect sizes of 0.68 [14] and 0.57 [15], and combining HRT and ERP [24]. The study was designed to
as separate scores for motor and vocal symptoms, as well compare the efficacy of therapeutic treatment based on the
as impairment [14] [effect sizes of 0.49 for motor tics, 0.50 same manual, delivered individually or in a group setting.
for vocal tics, and 0.57 for impairment]. A combination of The effectiveness was compared with the treatment results
HRT with mindfulness or cognitive strategies did not show shown in other studies.
additional benefit [16, 17]. Recent systematic reviews have
confirmed that HRT/CBIT are effective treatments in chil-
dren and adolescents with TS (SMD − 0.64, 95% CI − 0.99 Methods
to − 0.29; n = 133 [9, 10]). An overall moderate-to-large
treatment effect of behavior therapy in reduction of symp- Design
tom severity was suggested [7].
Exposure response prevention (ERP) in relation to tic The study was an open randomized controlled clinical trial
disorders is described in a manual for individual treat- for children and adolescents with Tourette syndrome or
ment of children and adolescents developed by Verdellen chronic motor or vocal tic disorder. The participants were
et al. [16]. In ERP, the patient is trained to endure the randomized to either individual training or group therapy.
premonitory urge to resist the tic symptoms. The partici- Both settings included eight sessions and an additional 9th
pants are exposed to stimuli that are known to elicit tics booster session. In the individual setting, the parents partici-
pated in the last 15 min of each session. In the group setting,

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the parents participated in the group at the end of the second, or attempts, primary severe anorexia nervosa. Furthermore,
fourth, eighth, and ninth session. Furthermore, the parents children and adolescents were excluded if their IQ was
were offered a group-based 2-h session of psycho-education. below 70, they had a lifetime diagnosis of pervasive devel-
The study was implemented at the specialized Tourette opmental disorder, or if they had been treated with HRT or
outpatient clinic at CAP, Risskov, Aarhus. All therapists ERP during the last 6 months. Patients were included in the
were qualified psychotherapists trained in HRT and ERP. study if sufficient treatment had been offered for a comorbid
The therapists were offered supervision. condition, including depression, severe ADHD or anorexia
nervosa, and where the tics symptoms still met the inclusion
Participants criteria. Children and adolescents on psychotropic medica-
tions for tics or other psychiatric disorders were included if
Inclusion started in November/December 2015 and was medication was stable with no planned changes during the
concluded in September 2017. Eligible participants were therapy period.
children and adolescents aged 9–17 years with a primary
diagnosis of either Tourette syndrome or a chronic motor/ Assessment methods and outcome measures
vocal tics disorder according to the WHO ICD-10 diag-
nostic criteria and the Diagnostic and Statistical Manual Diagnostic eligibility was established using a modified ver-
of Mental Disorders, Fourth edition, Text Revision, and of sion of the Schedule for Affective Disorders and Schizo-
moderate or greater severity corresponding to a total score phrenia for School-Age Children—Present and Lifetime ver-
on the Yale Global Tic Severity Scale (YGTSS) [25, 26] sion (K-SADS-PL) administered to the parents and child/
higher than 13 (higher than 9 if only motor or vocal tics were adolescent separately. The K-SADS-PL information was
described) [14] (Fig. 1). The inclusion and exclusion criteria used to confirm a primary diagnosis of chronic tic disorder
were chosen as to ensure that the study would be as close and to ensure that none of the exclusion criteria were met.
to the clinical practice as possible. Thus, exclusion criteria Furthermore, the parents were asked to provide background
included disorders that required immediate treatment: psy- information and to rate the CBCL, SDQ, and the sensory
chotic disorder, primary severe depression, suicidal ideation profile. The parents and the child/adolescent were asked to
provide specific information concerning the tic disorder.
An overview of the diagnostic instruments is provided in
N=102 eligible patients Table 1. There were no significant in-between differences
in the study period
in severity scores between time of primary assessment and
N=23, not TS as the start of treatment (baseline).
primary condition Primary outcome measures were the Yale Global Tics
N=18, age below 9 years Severity Scale (YGTSS) at session 8. Furthermore, the child/
The reminder not adolescent and the parents were asked to assess the global
motivated for therapy, or
the parents did not accept severity of tics on a 0–10 severity scale. Secondary outcome
the randomisation measures included Screen for Child Anxiety Related Emo-
tional Disorders (SCARED), the Mood and Feelings Ques-
tionnaire (MFQ). Finally, the child/adolescent completed
N=59 patients for
randomization the Premonitory Urge Scale (PUTS) and Beliefs About Tics
Scale (BATS) for analysis of the importance of the premoni-
tory urge and the thoughts and beliefs about tics. An over-
view of the scales is shown in Table 1.

N=31, individual setting N=28, group setting Schedule for Affective Disorders and Schizophrenia
for School‑Age Children—Present and Lifetime version
(K‑SADS‑PL)

A modified version of the Schedule for Affective Disor-

N=27, complete data N=27, complete data ders and Schizophrenia for School-Age Children—Present
achieved achieved and Lifetime version (K-SADS-PL). The K-SADS-PL is
a semi-structured diagnostic interview examining a range
of child psychopathology (age 7–17 years). In the present
Fig. 1  Flowchart for inclusion and exclusion of participants in the tic study, screening symptoms and supplemental symptoms
treatment study were used for selected diagnoses including depression,

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Table 1  An overview of the scales and instruments used at baseline and for evaluating treatment outcome
Instrument Assessment/baseline Treatment outcome Time of evaluation
Session number

Diagnostics instruments K-SADS [20]

CBCL [26]
Background information
Sensory profile [27]
Severity score YGTSS [23, 24] YGTSS 8th, 9th, FU
Global assessment scale Global assessment scale 8th, 9th
Other instruments SCARED [28–30] SCARED 4th, 8th, 9th, FU
MFQ [31–33] MFQ 4th, 8th, 9th, FU
Premonitory Urge Scale [34] Premonitory Urge Scale 4th, 8th, 9th, FU
Beliefs-scale [35] Beliefs-scale 4th, 8th, 9th, FU
Questions concerning the preferred method 8th
and best quality outcome

anorexia nervosa, ADHD, psychosis, OCD, and anxiety Sensory profile (SP)
disorders. The interview was conducted with both the par-
ents and the patients. In the present study, diagnoses of tic SP is a collection of questionnaires for different age groups
disorder or comorbidity were based on symptoms classi- The aim of the questionnaires is to assess children’s
fied as certain only. The K-SADS-PL has shown a good responses to commonly occurring sensory events and to
inter-rater reliability [27]. evaluate the ability to process the sensorimotor impressions.
SP includes 125 questions grouped into three main areas:
sensory processing, sensory modulation and behavior, and
Child Behavior Checklist (CBCL) emotional response. SP is standardized in the USA, using
1037 children with and without difficulties. The validity and
CBCL is a parent questionnaire evaluating a range of reliability of SP are acceptable [29].
behavioral and emotional problems in children and
adolescents. The questionnaire is used in the age range Yale Global Tics Severity Scale (YGTSS)
6–18 years. CBCL has 113 items rated on a three-point
scale (0 = not true; 1 = sometimes true; and 2 = often true). The YGTSS is a clinician-administered semi-structured
The results are depicted both in a total problem scale and interview including a checklist of all tics present in the past
several subscales. Test–retest reliability has been reported week. The YGTSS severity rating covers five dimensions
as 0.95–1.00, and the internal consistency as good to divided into ten items including the number of tics, fre-
excellent [28]. quency, intensity, complexity and interference of the motor
and vocal tics, and a separate evaluation of the functional
impairment. The scores are summed to yield separate motor
Background information and vocal tic scores (0–25) and a combined total tic score
(0–50). The functional impairment scale (range 0–50) is
As part of the general assessment at baseline, parents assessed, rating the tic-related disability over the past week.
were asked questions covering occupation and educational YGTSS has been shown to have high internal consistency
background as well as questions aimed at identifying the and stability [25, 26].
presence of parental psychopathology and any family
history of tic or other psychiatric and/or somatic disor- Global assessment scale
ders. Furthermore, both the patients and the parents were
asked questions aimed at determining the age of onset and The global assessment scale was developed for the present
describing preceding, reducing and exacerbating factors, study and is a Likert scale (0: no symptoms–10: maximum
as well as the general course of the disorder. Finally, the symptoms), where the child/adolescent makes a global
parents and the patients were asked to describe the per- assessment of the severity of the tic disorder, the frequency
sonal characteristics of the child. of the tic symptoms, and the intensity of the premonitory

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urge. The child/adolescent was asked to assess at baseline, Quality questions

and at session 8.
The same scale was used at every visit to evaluate the The child/adolescent is asked to answer questions con-
severity of every single tic symptom (the severity of the cerning which methods they prefer (HRT and/or ERP)
tic, the frequency of the tic, and the preceding premonitory and which changes they want to emphasize as the most
urge of the tics) important (effect on mood, tic intensity and impairment,
the belief of being able to control tics, attention, conflicts
with others, and/or self-esteem).
Screen for Child Anxiety Related Emotional Disorders
(SCARED)
Randomization and blinding
SCARED includes separate versions for parents and the
child/adolescent. It includes 41 items rated on a three- Randomization was performed in relation to the clinical
point scale and assesses the occurrence of anxiety symp- conference where a diagnosis of a chronic tic disorder
toms based on DSM-IV. Scores range from 0 to 82. The was confirmed. Furthermore, the exclusion criteria were
questionnaire is validated in children and adolescents (age evaluated. Randomization was performed using a stratified
8–18 years), and the internal consistency has proven excel- block randomization where every fourth diagnosed patient
lent [30–32]. alternately directed the following three patients to either
individual or group treatment. This procedure was chosen
to ensure as short a latency period as possible.
The Mood and Feelings Questionnaire (MFQ)
Evaluations of treatment response were made by an
independent evaluator who was not blinded to the treat-
MFQ assesses the occurrence of depressive symptoms, using
ment allocation, yet not involved in the treatment of the
13 items rated on a three-point scale [33]. Scores range
patient, and blinded to any previous evaluations. The
between 0 and 26, where high scores indicate a severe func-
evaluators were a specialized psychologist and a child
tional impairment [34]. The scale is prepared in separate ver-
and adolescent psychiatrist with several years of experi-
sions for children/adolescents (age 8–18 years) and parents.
ence in diagnosing, evaluating and treating tic disorders. A
The internal consistency is good [35].
random sample of 18 samples were audiotaped (10%) and
evaluated by another rater with extensive experience and
Premonitory Urge Scale (PUTS) expertise in the use of the YGTSS. The analysis revealed
that the intraclass correlation coefficient was 0.88 (95%
PUTS is a short self-reporting scale with nine items prepared CI 0.72–0.95) for Total Tics score (motor and vocal tics),
to measure the tic-related premonitory urge. The scale was and 0.89 (95% CI 0.74–0.95) for functional impairment.
developed by D. Woods, USA. The scale has proven consist-
ent and with a high stability [36]. The scale was translated
into Danish by the principal investigator (J. Nissen). After Treatments
a re-translation into English, the scale was approved by D.
Woods, USA. The therapeutic treatment for individual and group set-
ting was based on a newly developed manual [22] adapted
by the individual treatment manuals by Woods et al. [11]
Beliefs About Tics Scale (BATS) and Verdellen et  al. [18]. The newly designed manual
described a nine-session therapy (eight sessions and a
BATS is a self-reporting scale with 20 items developed to booster session) for either individual or group treatment.
assess the different beliefs children and adolescent expe- An overview of the treatment sessions is presented in
rience in relation to tic symptoms and to suppressing the Table 2. All participants trained in HRT for two sessions
tic symptoms. The scale was developed by A. Apter, Israel. (session 2 and 3) and in ERP for two sessions (session 4
Studies have shown a high validity in relation to the experi- and 5). In the following sessions, the participants were
ence of premonitory urge (PUTS) and to the severity and trained in both treatment modalities depending on the
functional impairment related to tics (YGTSS). The scale has presented symptoms. Thus, if the child for instance was
shown a high internal consistency [37]. The scale was trans- bothered with a certain tic at school, a competing response
lated into Danish by the principal investigator (J. Nissen). could be selected and trained for that particular tic. For
After a re-translation to English, the scale was approved by the remainder tic symptoms, ERP would be trained. The
Dr. Steinberg, Schneider Children’s Medical Center, Israel. content of the sessions was similar regarding individual

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Table 2  An overview of the

sessions Session 1 Psycho-education about tic disorders
Session 2 Introduction and training in HRT
Session 3 HRT continued. How to tell others about tics
Session 4 ERP introduction and training
Session 5 ERP continued. Comorbidity
Session 6 HRT and/or ERP training
Session 7 HRT and/or ERP training. Quiz: what do you now know about tics?
Session 8 HRT and/or ERP training. Relapse prevention
Session 9 HRT and/or ERP training. Relapse prevention. How to meet a new tic

treatment and group-based treatment whereby outcome The group session

measures were comparable.
The groups consisted of four participants and one thera-
Sessions pist. Every session lasted 2 h and the parents participated
in 20 min at the end of session two, four, eight, and nine.
All sessions consisted of a review of homework, of tic reduc-
ing or exaggeration factors or situations, review of tic symp- The individual treatment
toms and severity, premonitory urge intensity, review of the
applicable strategies to combat the tic symptoms and to control Every session lasted 1 h and the parents participated 15 min
premonitory urge, and the goals seen in relation to the home- at the end of every session.
work. At the end of the session, homework was assigned for
the child/adolescent, which also included a discussion of the Parent teaching
parent’s role in the training program to define the home assign-
ment for the parents. All parents were invited to participate in a 2-h teaching and
psycho-education held in groups with 10–20 participants.
HRT condition The focus for the teaching was

Each HRT session included a review of the specific tic symp- 1. What is a tic disorder?
toms with a detailed description of the premonitory urge, the 2. How to understand the occurrence of tics and the phe-
localization, and the course of the urge and the tic symptom. notypic presentation of tics.
HRT included awareness training, competing response defini- 3. Function-based assessment and intervention.
tion, detailed description and training, and social support. The 4. Psychosocial and therapeutic interventions with special
therapist-assisted practice was a key component of the HRT focus on HRT and ERP.
training. 5. Training at home—how can the parent support and
stimulate the training?
ERP condition
Statistical analysis
Each ERP session included a review of all tic symptoms with
a detailed description of the premonitory urge. ERP training Primary outcome measures included the YGTSS subscores
included awareness training, including the training of “just” and total score. Furthermore, the global assessment of sever-
looking at the premonitory urge, calmly and softly describing ity, frequency, and premonitory urge was examined.
the localization, the travel through the body, and the inten- Effect sizes from the present study were compared with
sity of the feeling. The child/adolescent was encouraged to results from both individual and group treatment studies.
describe how they could visualize the feeling in the body. The improvement at post-treatment assessment was tested
Again, the therapist-assisted practice in ERP training was a in each group using a paired t test. The effect sizes were
key component. calculated by a ratio of the mean difference and the standard

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deviation of the difference between baseline and follow-up occurrence of systematic differences was evaluated by the
(SD diff). Confidence interval (CI) of the effect sizes was average difference and confidence intervals. The random
computed using the non-parametric bootstrap with 100 repli- differences were quantified by the limit of agreement (95%
cations and normal-based standard error. Based on the study predicted intervals). The agreement between two raters
by Verdellen et al. [19], a combined effect size for ER and was accessed using the intraclass correlation coefficient
HRT was estimated as 1.24 and a combined ratio between (ICC).
the standard deviation of the difference between baseline Baseline characteristics and pre-treatment TS severity
­(SDdiff) and follow-up, and the pooled standard deviation scores were compared between the groups, using univari-
of baseline and follow-up ­SDwithin ­(SDdiff/SDwithin) was esti- ate Chi test for categorical variables and t tests for continu-
mated as 0.64. Using a study sample size of 60 (30 persons ous variables.
in each group), we expected to estimate an effect size in each Differences in TS scores were checked with respect to
group with a 95% confidence interval given by ± 0.23. the assumptions of normality using the normality plot and
The efficacy improvements were compared between homogeneity of variance using the F test. Significance was
the two treatment groups, using the unpaired t test. The defined as a p value of less than 0.05.

Table 3  Baseline characteristic
Individual setting (N = 31) Group setting (N = 28) Total group (N = 59) p

Age 12.30 (2.63) 12.18 (1.96) 12.24 (2.32) 0.85

Gender (male) 18 (58.1%) 19 (67.9%) 37 (62.7%) 0.45
Baseline YGTSS
 Total score 51.52 (13.04) 48 (12.12) 49.85 (12.62) 0.29
 Motor score 15.03 (3.57) 15.25 (3.33) 15.14 (3.43) 0.81
 Vocal score 9.16 (4.89) 8.43 (5.20) 8.81 (5.01) 0.58
 Total Tic score 24.19 (6.94) 23.68 (6.78) 23.95 (6.81) 0.77
 Functional impairment 27.39 (7.74) 24.25 (8.04) 25.90 (7.98) 0.13
SCARED
 Score by parents (N = 56) 21.04 (10.98) 22.71 (18.17) 21.88 (14.90) 0.68
 Score by children (N = 52) 22.61 (11.81) 26.08 (17.16) 24.21 (14.48) 0.39
MFQ
 Score by parents (N = 56) 5.79 (5.19) 5.75 (5.41) 5.77 (5.25) 0.98
 Score by children (N = 53) 4.36 (3.91) 5.12 (3.82) 4.72 (3.85) 0.48
 Occurrence of not-just-right [N (%)] 26/31 (83.9%) 15/28 (53.6%) 41/59 (69.5%) 0.018*
 Occurrence of OC symptoms [N (%)] 26/31 (83.9%) 16/28 (57.1%) 42/50 (71.2%) 0.024*
 Occurrence of premonitory urge [N (%)] 29/31 (93.5%) 25/28 (89.3%) 54/59 (91.5% 0.20
 CBCL total score (N = 58) 39.27 (25.39) 41.44 (25.96) 40.30 (25.46) 0.75
 CBCL score 1 10.43 (8.99) 12.11 (9.12) 11.24 (9.01) 0.48
 CBCL score 2 12.93 (9.15) 12.46 (10.07) 12.71 (9.52) 0.85
 CBCL score 3 5.33 (4.20) 5.07 (4.88) 5.21 (4.50) 0.83
 CBCL score4 7.57 (5.02) 6.96 (4.22) 7.28 (4.62) 0.62
 CBCL score 5 3.07 (3.14) 3.75 (3.11) 3.40 (3.12) 0.41
 CBCL score 6 3.2 (2.64) 3.18 (2.88) 3.19 (2.74) 0.98
 CBCL score 7 2.2 (2.76) 3.11 (2.91) 2.64 (2.85) 0.23
 Diagnosis of anxiety disorder [N (%)] 6/31 (19.4%) 4/28 (33.6%) 10/59 (16.9%) 0.53
 Diagnosis of ADHD [N (%)] 8/31 (25.8%) 12/28 (42.9%) 20/29 (33.9%) 0.038*
 Diagnosis of depression [N (%)] 1/31 (3%) 0/28 (0%) 1/59 (1.7%) 0.35
 Problems with structure and planning [N 3/31 (9.7%) 4/28 (14.3%) 7/59 (11.9%) 0.60
(%)] (ICD-10:DF83.9)

Total Tic score = sum motor score and vocal score, total = sum Total Tic score and functional impairment
*Significant difference between individual setting and group setting p < 0.05

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Results treatment (25.8%). Attrition in the group setting was 1/28

and individual setting 4/31. A total of N = 49 (83.1%)
Baseline characteristics reported a lifetime increased sensitivity, of which the
majority reported tactile hypersensitivity (N = 29, 59.2%)
102 adolescents were screened and 59 were randomly and increased sensitivity to noise and sounds (N = 35,
assigned to treatment in either an individual setting or 71.4%). N = 28 reported to have no strategies to work
group setting (Fig. 1). The age of the participants ranged against their tics. N = 26 had experience in suppressing
from 9 to 17 years (mean = 12.24, SD = 2.32) and N = 37 their tics at school, but experienced an increased intensity
(62.7%) were males. A total of N = 25 (42.4%) were treated when returning to their homes.
with medication, including methylphenidate (N = 10),
atomoxetine (N < 5), neuroleptics (N < 5), antidepressant Outcome
(N < 5) and melatonin (N = 5). Baseline characteristics are
shown in Table 3. There were no statistical between-group Mean scores, effect sizes, and confidence intervals on the
differences in relation to baseline severity scores, parental difference between individual and group setting for all study
and child scores for SCARED or MFQ, CBCL scores, or to outcomes are shown in Table 4. After eight sessions, there
the description of premonitory urge. However, individual was a decrease in the Yale Global Tics Severity total tic
participants reported a significantly increased tendency score of 9.48 (SD = 7.84) points for individual therapy and
to experience a not-just-right sensation, and reported 7.48 (SD = 5.44) points for group therapy showing effect
an increased number of OC symptoms. The most often sizes of 1.21 (0.79–1.63) and 1.38 (0.84–1.64), respectively.
reported OC symptoms were ritualized behaviors associ- Defining a responder level at 25% reduction of YGTSS total
ated with magic, order and symmetry, repeating behavior, tic score [38], 66.7% in both settings would be considered
and counting (66%). The content of OC symptoms was as responders (Table 4). There were no between-group dif-
comparable for the two treatment groups. Furthermore, ferences (individual versus group treatment), in total scores,
there was a significantly larger number of participants with motor scores, vocal scores, or in Total Tic scores on the
ADHD in the groups (42.9%) compared to the individual YGTSS. For the functional impairment score, a significantly
greater reduction was shown for the individual treatment

Table 4  Outcome after eight sessions

Pre [mean (SD)] Post [mean (SD)] Diff [mean (SD)] Effect size (± SD) PPI > 25% (%) p

Individual total 50.89 (12.46) 25.59 (10.04) 25.30 (15.24) 1.66 (1.11–2.21) 81.5 < 0.0001*
Individual motor 14.63 (3.56) 9.81 (3.44) 4.81 (4.10) 1.18 (0.71–1.64) 44.4 < 0.0001*
Individual vocal 9.15 (4.44) 4.48 (4.04) 4.67 (4.64) 1.01 (0.62–1.39) 63.0 < 0.0001*
Individual Total Tic score 23.78 (6.53) 14.30 (5.62) 9.48 (7.84) 1.21 (0.79–1.63) 66.7 < 0.0001*
Individual Functional impairment 27.19 (7.78) 10.93 (5.89) 16.26 (9.77) 1.66 (1.00–2.33) 92.6 <0.0001*
Group total 47.89 (12.33) 29.93 (13.33) 17.96 (11.34) 1.58 (0.99–2.18) 74.1 < 0.0001*
Group motor 15.22 (3.39) 10.52 (4.34) 4.70 (3.78) 1.24 (0.73–1.76) 48.1 < 0.0001*
Group vocal 8.19 (5.13) 5.41 (4.25) 2.78 (3.90) 0.71 (0.26–1.17) 51.9 0.001*
Group Total Tic score 23.41 (6.75) 15.93 (6.66) 7.48 (5.44) 1.38 (0.84–1.64) 66.7 < 0.0001*
Group functional impairment 24.41 (8.15) 13.89 (8.01) 10.52 (8.48) 1.24 (0.84–1.91) 70.4 <0.0001*
Total group total 49.39 (12.37) 27.76 (11.89) 21.63 (13.81) 1.57 (1.19–1.94) 77.8 < 0.0001*
Total group motor 14.93 (3.46) 10.17 (3.90) 4.76 (3.90) 1.22 (0.94–1.49) 46.3 < 0.0001*
Total group vocal 8.67 (4.77) 4.94 (4.14) 3.72 (4.35) 0.86 (0.58–1.13) 57.4 < 0.0001*
Total group Total Tic score 23.59 (6.58) 15.11 (6.16) 8.48 (6.76) 1.26 (0.94–1.57) 66.7 <0.0001*
Total group functional impairment 25.80 (8.01) 12.41 (7.12) 13.39 (9.51) 1.41 (1.00–1.82) 81.5 < 0.0001*
Global impairment 7.31 (1.55) 4.26 (2.30) 3.05 (2.15) < 0.0001*
Global frequency 7.23 (1.78) 4.46 (2.38) 2.77 (3.05) < 0.0001*
Global stress 6.85 (1.90) 3.51 (2.21) 3.33 (2.26) < 0.0001*
Global urge 5.44 (3.10) 3.85 (2.58) 1.59 (3.81) 0.013*

Completer sample (N = 54). Means and differences (pre- and post-treatment scores) (SD), effect sizes, and the percentages of patients who
improved > 25% (PPI > 25%). Total Tic score = sum motor score and vocal score, total = sum Total Tic score and functional impairment
*Significance p < 0.05

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European Child & Adolescent Psychiatry (2019) 28:57–68 65

Fig. 2  Total Tic score and

functional impairment score
from baseline to eighth session
for each of individual therapy
and group setting. *p < 0.05
significant score reductions
from baseline to eighth session.
YGTSS Yale Global Tics Sever-
ity Scale

Fig. 3  PUTS scores and BATS

scores at baseline, fourth, and
eighth session for the indi-
vidual therapy and group setting
combined. *p < 0.05 significant
score reductions from baseline
to eighth session

(p = 0.025) (Fig. 2). Likewise, the reductions in global fre- urge (at baseline: rho 0.78 (0.25–1.30), p = 0.005, and at
quency and global stress were rated greater in the individual session 8: rho 0.87 (0.27–1.46), p = 0.005).
treatment compared to group treatment. The BATS scale showed a significant total reduction
Premonitory urge (evaluated by PUTS) and belief about of 7.74 (4.79–10.70) (p = 0.0001) point score which was
the tics (evaluated by BATS) were measured at baseline, ses- significantly larger in the individual group compared to
sion 4, and session 8. There was no significant change in the group treatment (p = 0.0075) (Fig. 3). The PUTS scale and
total PUTS scores during treatment (Fig. 3). The PUTS score the BATS scale were positively associated both at base-
correlated with the assessment of the global premonitory line [rho 0.22 (0.07–0.38), p = 0.005], at session 4 [rho

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66 European Child & Adolescent Psychiatry (2019) 28:57–68

0.40 (0.26–0.53), p = 0.0001], and at session 8 [rho 0.31 may experience a better opportunity to discuss the influence
(0.18–0.44), p = 0.0001]. on the family. These circumstances may influence the gen-
Parents reported a significant reduction in scores meas- eral functional impairment experienced by both the child and
ured on SCARED (p = 0.0019), which could not be shown the family. For both the individual and groups setting, most
in the children’s ratings. There were no between-group dif- of the participants reported to have experienced achieving a
ferences. No differences could be shown for MFQ scores. subjective feeling of having control or reduced tics intensity.
N = 9 (15.3%) of the participants reported that they had Thus, several participants reported that they might still have
preferred HRT compared to N = 19 (32.2%) who had pre- tics, but that the tics had become less restrictive for their
ferred ERP, and N = 16 (27.1%) reported the use of both lives. The finding that group HRT/ERP is an effective treat-
methods. A subjective feeling of having control (N = 36) or ment validates the only other study that examines the effect
a reduced tic intensity (N = 30) was the most often reported of group treatment (HRT versus educational treatment [21]).
effects of the training program. Sensory phenomena are very frequent in TS, premonitory
urges being one of the most often reported preceding sensa-
tions [36]. In children, however, a developmental aspect has
Discussion to be considered since younger children rarely experience
a premonitory urge [39]. In the present study, PUTS scores
This is the first Scandinavian study evaluating the effec- showed no difference from baseline to session 8 even though
tiveness of a newly developed manual combining ERP and tic severity was reduced. In a study from 2013, Specht et al.
HRT [24]. To our knowledge, it is also the first randomized showed that urge ratings did not show an increase during the
controlled study that compares the efficacy of therapeutic initial periods of tic suppression, or a decline in urge inten-
treatments based on the same manual delivered individually sity during the following prolonged tic suppression [40].
or in a group setting. Correspondingly, Ganos et al. found no correlation between
Compared to other studies, the present study shows that scores of premonitory urges and the ability to suppress tics
treatment combining HRT and ERT training is effective in [13]. Furthermore, the premonitory urge has been shown
both groups and as an individual treatment. The decrease to remain unchanged during tic suppression [12, 41]. Thus,
from baseline to end point on the Total Tic score (motor and some patients may not experience a habituation to the pre-
vocal tic score) of YGTSS of 9.48 points (individually) and monitory urge, but rather have to learn to accept and endure
7.48 points (group setting) (8.48 points for all participants) the urge feeling. There was no difference in change of PUTS
is comparable or slightly greater than the effects shown in scores between individual and group setting. The PUTS
previous studies [13, 19]. There was no significant difference score correlated at all time points with the BATS score. The
in Total Tic score between individual and group treatment. BATS score was reduced significantly from treatment start
Children and adolescents were trained in both HRT and to end point, suggesting that tics treatment had a significant
ERP, which gave them a possibility to alternate between the impact on thoughts and interpretations of tics. Furthermore,
strategies depending on their general situation. In both the the scores of the BATS were significantly more reduced dur-
group setting and in individual therapy, a substantial number ing the individual therapy compared to the group setting,
of the participants described that they used a combination suggesting that individual treatment is more likely to have
of the methods using HRT for certain selected tics and ERP an indirect effect on the children’s interpretations of their tic
for training against all tics. Defining a 25% reduction on the disorder. To our knowledge, this is the first study compar-
YGTSS Total Tic score as predictive for a positive response ing the influence of group or individual treatment on BATS
[38], 66.7% of the participants were rated as responders. scores in children and adolescents with Tourette syndrome.
Verdellen et al. described the percentage of patients who Parents rated a significant decrease in anxiety scores
improved more than 30% [19]. They showed that 58% of measured by SCARED, whereas the children did not report
patients in the ER group and 28% in the HR showed a reduc- a change. Similarly, no change was reported on the MFQ
tion that exceeded 30%. In the present study, a reduction of scale. There were no in-between-group differences.
more than 30% was shown for 59.3% of participants. Our results have several clinical implications. First, the
The participants reported a significant reduction in the efficacy of a combined treatment of HRT and ERP in both an
functional impairment score, which was significantly greater individual and group setting expands the available treatment
in the individual setting. Also, measured by the subjective possibilities for tic disorders in children and adolescents.
global scores, the individual training showed the greatest The participants represented a clinical sample with few
outcomes. In an individual setting, the interaction between exclusion criteria; thus the manual has a broad applicabil-
the therapist and the child may become more immediate, and ity. The efficacy shown in the present study is comparable
the therapist is able to focus more intensely on a particular with those found in medication treatment studies [42, 43],
child’s resources and difficulties. Furthermore, the parents further increasing the argument for a therapeutic treatment

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European Child & Adolescent Psychiatry (2019) 28:57–68 67

in preference to medication. Secondly, the children achieved 3. Cath DC, Hedderly T, Ludolph AG, Stern JS, Murphy T, Hart-
an experience of enhanced control of their tic disorder, and mann A, Czernecki V, Robertson MM, Martino D, Munchau A,
Rizzo R, ESSTS Guidelines Group (2011) European clinical
the study shows an important influence on the interpretation guidelines for Tourette syndrome and other tic disorders. Part I:
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come of individual therapy to the outcome of group ther- 7-016-0823-8
apy. The methods that were combined in the present study, 5. Verdellen C, van de Griendt Hartmann A, Murphy T, ESSTS
Guidelines Group (2011) European clinical guidelines for Tourette
HRT and ERP, are well-established treatment methods in an syndrome and other tic disorders. Part III: behavioural and psy-
individual setting with effect sizes comparable to those pre- chosocial interventions. Eur Child Adolesc Psychiatry 20(4):197–
sented in the present study. Thus, even though effect sizes for 207. https​://doi.org/10.1007/s0078​7-011-0167-3
the combined treatment are comparable with effect sizes for 6. Roessner V, Plessen KJ, Rothenberger A, Ludolph AG, Rizzo
R, Skov L, Strand G, Stern JS, Termine C, Hoekstra PJ, ESSTS
either HRT or ERP, the combination renders the child able Guidelines Group (2011) European clinical guidelines for Tou-
to choose the suitable method depending on the tic symptom rettes syndrome and other tic disorders. Part II: pharmacological
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7. McGuire JF, Ricketts EJ, Piacentini J, Murphy TK, Storch EA,
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outcome data. Further research into the durability of the 40150​0635
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treatment effect is warranted. ing nervous habits and tics. Behav Res Ther 11:619–628
9. Hollis C, Pennant M, Cuenca J, Glazebrook C, Kendall T, Whit-
Funding  The study was performed during clinical practice. PI Judith tingyon C, Stockton S, Larsson L, Bunton P, Dobson S, Groom
Nissen was partly supported by the Lundbeck Foundation, Grant num- M, Hedderly T, Heyman I, Jackson GM, Jackson S, Murphy T,
ber R185-2014-2486. Rickards H, Roberson M, Stern J (2016) Clinical effectiveness
and patient perspectives of different treatment strategies for
Compliance with ethical standards  tics in children and adolescents with Tourette syndrome: a sys-
tematic review and qualitative analysis. Health Technol Assess
Conflict of interest  The authors declare that they have no competing 20(4):1366–5278. https​://doi.org/10.3310/hta20​040
interests. 10. Whittington C, Pennant M, Kendall T, Grazebrook C, Trayner P,
Groom M, Hedderly T, Heyman I, Jackson G, Jackson S, Mur-
phy T, Rickards H, Robertson M, Stren J, Hollis C (2016) Prac-
Ethics approval  The study was approved by the National Ethical Com- titioner review: treatments for Tourette syndrome in children and
mittee (1-10-72-216-15) and the Danish Data Protection Agency (1-16- young people—a systematic review. J Child Psychol Psychiatry
02-490-15). 57(9):988–1004. https​://doi.org/10.1111/jcpp.12556​
11. Woods DW, Piacentini JC, Chang SW, Deckersbach T, Ginsberg
Informed consent  Oral and written information was given to parents GS, Peterson AL, Scahill LD, Walkup JT, Wilhelm S (2003) Man-
and patients, and written consent from patients over 15 years and par- aging Tourette syndrome: a behavioral intervention for children
ents was received. and adults. University Press, Oxford
12. Houghton DC, Capriotti MR, Scahill LD, Wilhelm S, Peterson
Open Access  This article is distributed under the terms of the Crea- AL, Walkup JT, Piacentini J, Woods DW (2017) Investigating
tive Commons Attribution 4.0 International License (http://creat​iveco​ habituation to premonitory urges in behavior therapy for tic dis-
mmons​.org/licen​ses/by/4.0/), which permits unrestricted use, distribu- orders. Behav Ther 48(6):834–846. https​://doi.org/10.1016/j.
tion, and reproduction in any medium, provided you give appropriate beth.2017.08.004
credit to the original author(s) and the source, provide a link to the 13. Ganos C, Kahl U, Schunke O, Kühn S, Haggard P, Gerloff C,
Creative Commons license, and indicate if changes were made. Roesner V, Thomalla G, Münchau A (2012) Are premonitory
urges a prerequisite of tic inhibition in Gilles de la Tourette syn-
drome? J Neurol Neurosurg Psychiatry 83(10):975–978. https​://
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